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      Intractable hiccups after VNS implantation: a case report

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          Abstract

          Background

          Hiccups (medically termed, “singultus”), when intractable, can cause significant medical consequences such as aspiration, malnutrition, and depression, leading to poor quality of life. Several case reports have shown that vagus nerve stimulator (VNS) implantation can help treat central idiopathic intractable hiccups. However, we present a contrary case of a patient who developed intractable singultus following VNS placement for medically refractory epilepsy.

          Case presentation

          We report a 71-year-old male patient with drug-resistant epilepsy who underwent VNS implantation and developed intractable hiccups shortly thereafter. The hiccups were severe and persistent, such that the patient developed a Mallory-Weiss tear, which required intensive care, invasive intubation and mechanical ventilation, and a prolonged rehabilitation course. Despite multiple therapies including phrenic nerve block and Nissen fundoplication, the patient’s hiccups persisted and only stopped once the VNS was permanently deactivated.

          Conclusions

          Little is known about the incidence of hiccups after VNS implantation. We present one case of hiccups as a direct consequence of VNS implantation. The clinical impact of this report is significant given the relative unfamiliarity of hiccups as an adverse effect of VNS implantation. Neurologists and epileptologists, who present VNS implantation as a surgical option for seizure control to their patients, should be aware of the possibility of singultus development and its significant physical and emotional ramifications.

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          Most cited references34

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          Vagus Nerve Stimulation.

          The vagus nerve is a major component of the autonomic nervous system, has an important role in the regulation of metabolic homeostasis, and plays a key role in the neuroendocrine-immune axis to maintain homeostasis through its afferent and efferent pathways. Vagus nerve stimulation (VNS) refers to any technique that stimulates the vagus nerve, including manual or electrical stimulation. Left cervical VNS is an approved therapy for refractory epilepsy and for treatment resistant depression. Right cervical VNS is effective for treating heart failure in preclinical studies and a phase II clinical trial. The effectiveness of various forms of non-invasive transcutaneous VNS for epilepsy, depression, primary headaches, and other conditions has not been investigated beyond small pilot studies. The relationship between depression, inflammation, metabolic syndrome, and heart disease might be mediated by the vagus nerve. VNS deserves further study for its potentially favorable effects on cardiovascular, cerebrovascular, metabolic, and other physiological biomarkers associated with depression morbidity and mortality.
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            Systemic review: the pathogenesis and pharmacological treatment of hiccups.

            Hiccups are familiar to everyone, but remain poorly understood. Acute hiccups can often be terminated by physical manoeuvres. In contrast, persistent and intractable hiccups that continue for days or months are rare, but can be distressing and difficult to treat.
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              Chronic Hiccups: An Underestimated Problem.

              Persistent singultus, hiccupping that lasts for longer than 48 hours, can have a tremendous impact on a patient's quality of life. Although involved neurologic structures have been identified, the function of hiccups remains unclear-they have been controversially interpreted as a primitive reflex preventing extent swallowing of amniotic fluid in utero, an archaic gill ventilation pattern, or a fetus' preparation for independent breathing. Persistent singultus often presents as a symptom for various diseases, most commonly illnesses of the central nervous system or gastrointestinal tract; they can also be evoked by a variety of pharmacological agents. It is often impossible to define a singular cause. A wide range of treatment attempts, pharmacological and nonpharmacological, have been concerted to this date; however, chlorpromazine remains the only Food and Drug Administration-approved drug in this context. Large-scale studies on efficacy and tolerance of other therapeutic strategies are lacking. Gabapentin, baclofen, and metoclopramide have been reported to accomplish promising results in reports on the therapy of persistent singultus; they may also be effective when given in combination with other drugs, eg, proton pump inhibitors, or as conjoined therapy. As another approach of note, acupuncture treatment was able to abolish hiccups in a number of studies. When managing hiccup patients within the clinical routine, it is of importance to conduct a comprehensive and effective diagnostic workup; a well-functioning interdisciplinary team is needed to address possible causes for the symptom. Persistent singultus is a medical problem not to be underestimated; more research on options for effective treatment would be greatly needed.
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                Author and article information

                Contributors
                myriama@bu.edu
                Journal
                BMC Neurol
                BMC Neurol
                BMC Neurology
                BioMed Central (London )
                1471-2377
                10 August 2023
                10 August 2023
                2023
                : 23
                : 297
                Affiliations
                GRID grid.189504.1, ISNI 0000 0004 1936 7558, Department of Neurology, , Boston Medical Center/Boston University Chobanian & Avedisian School of Medicine, ; Boston, MA 02118 USA
                Article
                3352
                10.1186/s12883-023-03352-x
                10413770
                2a8c423c-2d20-4439-975d-03f98b939a8a
                © BioMed Central Ltd., part of Springer Nature 2023

                Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data.

                History
                : 12 April 2023
                : 28 July 2023
                Categories
                Case Report
                Custom metadata
                © BioMed Central Ltd., part of Springer Nature 2023

                Neurology
                intractable hiccups,vagus nerve stimulator,singultus,neuromodulation,epilepsy,case report
                Neurology
                intractable hiccups, vagus nerve stimulator, singultus, neuromodulation, epilepsy, case report

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